Polarity proteins control ciliogenesis via kinesin motor interactions

Shuling Fan, Toby W Hurd, Chia-Jen Liu, Samuel W Straight, Thomas Weimbs, Elizabeth A Hurd, Steven E Domino, Ben Margolis

Research output: Contribution to journalArticlepeer-review


BACKGROUND: Cilia are specialized organelles that play a fundamental role in several mammalian processes including left-right axis determination, sperm motility, and photoreceptor maintenance. Mutations in cilia-localized proteins have been linked to human diseases including cystic kidney disease and retinitis pigmentosa. Retinitis pigmentosa can be caused by loss-of-function mutations in the polarity protein Crumbs1 (CRB1), but the exact role of CRB1 in retinal function is unclear.

RESULTS: Here we show that CRB3, a CRB1-related protein found in epithelia, is localized to cilia and required for proper cilia formation. We also find that the Crumbs-associated Par3/Par6/aPKC polarity cassette localizes to cilia and regulates ciliogenesis. In addition, there appears to be an important role for the polarity-regulating 14-3-3 proteins in this process. Finally, we can demonstrate association of these polarity proteins with microtubules and the microtubular motor KIF3/Kinesin-II.

CONCLUSIONS: Our findings point to a heretofore unappreciated role for polarity proteins in cilia formation and provide a potentially unique insight into the pathogenesis of human kidney and retinal disease.

Original languageEnglish
Pages (from-to)1451-61
Number of pages11
JournalCurrent biology : CB
Issue number16
Publication statusPublished - 24 Aug 2004


  • 14-3-3 Proteins
  • Animals
  • Blotting, Western
  • Calcium-Binding Proteins
  • Cell Polarity
  • Cells, Cultured
  • Cilia
  • Dogs
  • Electrophoresis, Polyacrylamide Gel
  • Immunohistochemistry
  • Kidney
  • Kinesin
  • Male
  • Membrane Glycoproteins
  • Mice
  • Microtubules
  • Muscle Proteins
  • Mutation
  • Oligonucleotides
  • Precipitin Tests
  • Protein Kinase C
  • Spermatozoa
  • Tyrosine 3-Monooxygenase

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